Cerebral palsy is an umbrella term for a group of

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1 ONLINE ONLY Severity of malocclusion in patients with cerebral palsy: Determinant factors Cristina Batista Miamoto, a Maria Letıcia Ramos-Jorge, b Luciano José ereira, a Saul Martins aiva, c Isabela Almeida ordeus, c and Leandro Silva Marques a Tr^es Coraç~oes, Diamantina, and Belo Horizonte, Minas Gerais, Brazil Introduction: Our aims in this study were to compare the biopsychosocial aspects of patients with cerebral palsy and subjects in a control group, establish the severity of malocclusion, and identify determinant factors. Methods: The group with cerebral palsy included 60 patients diagnosed with the spastic form of the disease. The control group included 60 randomly selected healthy subjects with various malocclusions. Data were collected through questionnaires, medical charts, and clinical evaluations. The criteria of the dental aesthetic index were used for the diagnosis of malocclusion. Comparisons between groups and between the independent variables and dependent variable (severity of malocclusion) were performed by using the chi-square test ( #0.05) and multivariate logistic regression (forward stepwise procedure). Results: Significant differences between the groups were found for these variables: tooth loss, overjet, anterior open bite, facial type, breathing pattern, drooling, difficulty in swallowing, and lip incompetence. Conclusions: The main risk factors associated with the severity of malocclusion were cerebral palsy, mouth breathing, lip incompetence, and long face. (Am J Orthod Dentofacial Orthop 2010;138:394.e1-394.e5) Cerebral palsy is an umbrella term for a group of conditions characterized essentially by motor dysfunctions that might be associated with sensory or cognitive impairment stemming from a nonprogressive brain lesion during development. 1 Its prevalence is about 2 of every 1000 live births. 1-3 Impairments range from mild, with little difficulty, to severe, in which the child completely depends on others for the activities of daily living. The classification of cerebral palsy depends on the predominant motor alteration. Spastic cerebral palsy, the most common type, is characterized by a lesion in the cerebral cortex, with reduction in strength and increase in muscle tonus. Athetoid cerebral palsy is characterized by involuntary movements. Ataxic cerebral palsy is characterized by difficulties in motor coordination (trembling when performing a movement). The a rofessor, Department of Orthodontics, University of Vale do Rio Verde (UNINCOR), Tr^es Coraç~oes, Brazil; private practice, Diamantina, Brazil. b rofessor, Department of ediatric Dentistry, Universidade Federal dos Vales do Jequitinhonha e Mucuri, Diamantina, Minas Gerais, Brazil. c rofessor, Department of ediatric Dentistry, Universidade Federal de Minas Gerais, Belo Horizonte, Minas Gerais, Brazil. The authors report no commercial, proprietary, or financial interest in the products or companies described in this article. Reprint requests to: Leandro Silva Marques, Rua Arraial dos Forros 215, Diamantina, Minas Gerais, Brazil, CE: ; , lsmarques21@ yahoo.com.br. Submitted, September 2009; revised and accepted, March /$36.00 Copyright Ó 2010 by the American Association of Orthodontists. doi: /j.ajodo mixed type involves characteristics of 2 types of cerebral palsy at the same time (eg, spastic and athetoid). 3 In relation to oral health, persons with cerebral palsy are more prone to problems. They have greater prevalence of enamel hypoplasia, high frequencies of carbohydrate and pasty food intake, self-hygiene challenges, greater difficulty in chewing and swallowing, greater use of medication, and greater prevalences of periodontal disease and malocclusion. 2,4-6 The main characteristics associated with the prevalence of malocclusion among patients with cerebral palsy are musculoskeletal abnormalities, altered cranial-base relationships, premature tooth eruptions, and lip incompetence. 7 However, the severity of malocclusion varies, depending on the degree of impairment. A critical evaluation of the literature showed that studies failed to identify determinant factors in considering the severity of malocclusion in those with cerebral palsy. 2,6-12 The methods and statistical analyses used were limited to providing descriptive data and comparisons between heterogeneous groups but failed to consider diverse biopsychosocial variables. This problem is all the more critical because children with cerebral palsy receive little dental care, mainly because the difficulty in handling them. 2-4 Thus, a better understanding of the factors related to the prevalence and severity of malocclusion in these patients is essential to planning actions and public policies for the promotion of oral health. Our aims in this study were to compare the biopsychosocial aspects of subjects with cerebral palsy with 394.e1

2 394.e2 Miamoto et al American Journal of Orthodontics and Dentofacial Orthopedics October 2010 those of persons in a control group, establish the severity of malocclusion, and identify determinant factors. MATERIAL AND METHODS The sample included 120 subjects (60 with cerebral palsy, 28 boys and 32 girls; and 60 with no physical or mental impairment, 19 boys and 41 girls) with an average age of 12.6 years. Those with cerebral palsy had the spastic form, with little or no control over their arms and legs, no ability to execute activities without assistance, and mental impairment. The control group was selected randomly from healthy people with various malocclusions who awaited orthodontic treatment at University of Itaúna. The inclusion criteria for participation in the group with cerebral palsy were a confirmed diagnosis of spastic cerebral palsy and no previous orthodontic intervention. arents or guardians of the participants signed informed consent forms. The study was approved by the ethics committee of the University of Tr^es Coraç~oes in Brazil. The data were collected through questionnaires, medical charts, and clinical examinations of the participants. The criteria of the dental aesthetic index (DAI) were used for the diagnosis of malocclusion. The DAI provides 4 outcome possibilities: mild malocclusion or absence of abnormality, the treatment of which is unnecessary (DAI \25); defined malocclusion, the treatment of which is elective (DAI ); severe malocclusion, the treatment of which is highly desirable (DAI ); and very severe or debilitating malocclusion, the treatment of which is fundamental (DAI.36). 13 Because all the participants in this study had malocclusions, this variable was dichotomized into moderate malocclusion (DAI #30) and severe malocclusion (DAI.30). Crossbite and facial type were also determined. The malocclusions were clinically classified as Angle Class I, Class II, or Class III. Lip incompetence was assessed with the method described by Ballard: mandible in physiological resting position in juxtaposition (sealed), with no contraction of the orbicular muscles of the mouth or mentalis. 14 Lip incompetence and breathing type (nasal or mouth) were determined during the clinical examination and the interview with the parent or guardian when the child thought he or she was not being observed. Drooling was determined based on the method described by Thomas-Stonell and Greenberg 15 : 1, no drool; 2, medium: small amount, only on the lips; 3, moderate: small amount on lips and chin; 4, severe: drool on clothes; and 5, profuse: wet clothes, hands, and objects. Cerebral palsy was diagnosed based on the medical chart. Table I. Univariate analysis considering aspects related to malocclusion between the cerebral palsy and control groups Control, n (%) Cerebral palsy, n (%) Tooth loss None 59 (55.1) 48 (44.9) At least 1 1 (7.7) 12 (92.3) Crowding None 31 (43.1) 41 (56.9) or 2 segments 29 (60.4) 19 (39.6) Spacing None 34 (54.0) 29 (46.0) or 2 segments 26 (45.6) 31 (54.4) Diastema (mm) \2 47 (54.7) 39 (45.3) $2 13 (38.2) 21 (61.8) Maxillary irregularity (mm) \2 48 (47.5) 53 (52.5) $2 12 (63.2) 7 (36.8) Mandibular irregularity (mm) \2 50 (47.6) 55 (52.4) $2 10 (66.7) 5 (33.3) Overjet (mm) \4 46 (63.9) 26 (36.1) \0.001 $4 14 (29.2) 34 (70.8) Mandibular protrusion No 59 (50.4) 58 (49.6) Yes 1 (33.3) 2 (66.7) Anterior open bite (mm) \2 57 (62.6) 34 (37.4) \0.001 $2 3 (10.3) 26 (89.7) osterior crossbite Absent 52 (51.5) 49 (48.5) resent 8 (42.1) 11 (57.9) Average 45 (61.6) 28 (38.4) Short face 1 (50.0) 1 (50.0) Long face 14 (31.1) 31 (68.9) Angle classification Class I 31 (56.4) 24 (43.6) Class II 25 (53.2) 22 (46.8) Class III 4 (22.2) 14 (77.8) Comparisons between groups and between the independent and dependent (severity of malocclusion) variables were carried out by using the chi-square test ( #0.05). Variables with a value #0.20 were included in the multivariate logistic regression model (forward stepwise procedure). RESULTS Concerning the occlusal problems, the patients with cerebral palsy had significant differences compared with the control group for tooth loss, overjet, and anterior open bite. Differences between the groups were also

3 American Journal of Orthodontics and Dentofacial Orthopedics Miamoto et al 394.e3 Volume 138, Number 4 found for facial type and Angle classification (Table I). Table II shows severe malocclusions in 68% of the patients with cerebral palsy, in 80.6% of the mouth breathers, in 84.2% of those with severe drooling, in 87.3% of those with difficulty swallowing, in 74.4% of those with lip incompetence, and in 71.1% of those with long face. After adjusting the model, the logistic regression analysis showed that those with cerebral palsy (3.21 [ ]), mouth breathers (4.82 [ ]), those with lip incompetence (2.86 [ ]), and those with long face (5.48 [ ]) had a greater chance of having a severe malocclusion (Table III). DISCUSSION atients with cerebral palsy are classified by the clinical aspects of muscle tonus abnormalities and the type of movement disorder: spastic, athetoid, ataxic, and mixed. 3 Since the patients in this study had the spastic form, with severe physical and mental impairment, comparisons with other studies should be made with caution. The results support the hypothesis that malocclusions are more prevalent and severe in patients with cerebral palsy than in healthy subjects. A number of studies reported greater prevalences of malocclusion in those with cerebral palsy compared with control groups. 6,7,9-12 The manifestation of malocclusion in this population has been attributed to the low tonicity of facial muscles and the uncoordinated movements of the lips and tongue. 6,16,17 However, the methodologies and statistical analyses used in previous studies were limited to descriptive data and comparisons between groups without considering biopsychosocial variables. In this study, overjet and anterior open bite were significantly more prevalent in the patients with cerebral palsy; this suggests an association with muscle impairment. Because the tone and function of the orofacial muscles with cerebral palsy can be abnormal, these children s facial growth and occlusion might be outside normal limits. Thus, is seems reasonable that lip incompetence might have some relevance in the etiology of increased overjet in children with cerebral palsy and with respect to a drooling habit. 6 Another finding related to occlusal characteristics is greater tooth loss among patients with cerebral palsy; this certainly contributes toward the greater prevalence of malocclusion. Children with cerebral palsy have high rates of gingival problems and caries, especially in the deciduous dentition. 2,4 Few professionals are technically trained to treat these patients, because this information is virtually omitted or incomplete in university courses. Thus, these patients receive little dental care. Table II. Univariate analysis between biopsychosocial variables and severity of malocclusion Severity of malocclusion (DAI) Moderate Severe Control 47 (78.3) 13 (21.7) \0.001 Cerebral palsy 19 (31.7) 41 (68.3) Sex Female 45 (61.6) 28 (38.4) Male 21 (44.7) 26 (55.3) Age (y) \10 32 (57.1) 24 (42.9) $10 34 (53.1) 30 (46.9) Birth weight (g) (54.7) 24 (45.3) # (50.0) 11 (50.0) remature birth No 58 (58.6) 41 (41.4) Yes 7 (36.8) 12 (63.2) Schooling of caregiver (y).8 36 (62.1) 22 (37.9) #8 30 (48.4) 32 (51.6) Common old, last time (mo) $1 34 (50.7) 33 (49.3) \1 13 (50.0) 13 (50.0) Breathing Nasal 59 (70.2) 25 (29.8) \0.001 Mouth 7 (19.4) 29 (80.6) Lip incompetence No 55 (71.4) 22 (28.6) \0.001 Yes 11 (25.6) 32 (74.4) Drool Absent 60 (66.7) 30 (33.3) \0.001 Moderate 3 (27.3) 8 (72.7) Severe 3 (15.8) 16 (84.2) Difficulty swallowing No 64 (61.5) 40 (38.5) \0.001 Yes 2 (12.5) 14 (87.5) Dentition Deciduous 5 (38.5) 8 (61.5) Mixed 42 (61.8) 26 (38.2) ermanent 19 (48.7) 20 (51.3) Average 53 (72.6) 20 (27.4) \0.001 Short face 0 (0.0) 2 (100.0) Long face 13 (28.9) 32 (71.1) The main risk factors associated with malocclusion severity in this study were cerebral palsy, mouth breathing, lip incompetence, and long face. These findings have considerable clinical significance, since patients with cerebral palsy and serious mental impairment normally also have mouth breathing, lip incompetence, and long face. Severe malocclusion can trigger conditions such as temporomandibular joint disorders and problems ingesting food. 18 This makes the treatment of these patients all the more challenging and necessary.

4 394.e4 Miamoto et al American Journal of Orthodontics and Dentofacial Orthopedics October 2010 Table III. Multiple logistic regression analysis between independent variables and severity of malocclusion (final model) Unadjusted odds ratio (CI 95%) Adjusted odds ratio (CI 95%) Control Cerebral palsy 7.80 ( ) \ ( ) Sex Female Male 1.99 ( ) ( ) Schooling of caregiver (y) # ( ) ( ) remature birth No 1.00 Yes 2.42 ( ) Common cold, last time (mo) $ \ ( ) Difficulty swallowing No 1.00 Yes ( ) Lip incompetence No Yes 7.27 ( ) \ ( ) Drool Absent 1.00 Moderate 5.33 ( ) Severe ( ) \0.001 Type of breathing Nasal Mouth 9.78 ( ) \ ( ) Dentition Deciduous 1.00 Mixed 0.39 ( ) ermanent 0.66 ( ) Average Short face 4.31 ( ) ( ) Long face 6.52 ( ) \ ( ) Orthodontists must recognize their role on the multidisciplinary team (speech therapy, psychology, ophthalmology, orthopedics, pediatrics, neurology, nutrition, and so on) that is responsible for these patients, beginning at an early age. Early, multidisciplinary treatment can help to minimize the harmful effects of malocclusion and improve the quality of life of these patients. CONCLUSIONS The main factors associated with severe malocclusion were cerebral palsy, mouth breathing, lip incompetence, and long face. atients with cerebral palsy also had a greater prevalence of malocclusion compared with healthy subjects. REFERENCES 1. Gupta R, Appleton RE. Cerebral palsy: not always what it seems. Arch Dis Child 2001;85: Mitsea AG, Karidis AG, Donta-Bakoyianni C, Spyropoulos ND. Oral health status in Greek children and teenagers, with disabilities. J Clin ediatr Dent 2001;26: Surabian SR. Developmental disabilities: epilepsy, cerebral palsy, and autism. J Calif Dent Assoc 2001;29: dos Santos MT, Masiero D, Simionato MR. Risk factors for dental caries in children with cerebral palsy. Spec Care Dentist 2002;22: Guare Rde O, Ciampioni AL. revalence of periodontal disease in the primary dentition of children with cerebral palsy. J Dent Child (Chic) 2004;71: Franklin DL, Luther F, Curson MEJ. The prevalence of malocclusion in children with cerebral palsy. Eur J Orthod 1996;18:

5 American Journal of Orthodontics and Dentofacial Orthopedics Miamoto et al 394.e5 Volume 138, Number 4 7. Winter K, Baccaglini L, Tomar S. A review of malocclusion among individuals with mental and physical disabilities. Spec Care Dentist 2008;28: Carmagnani FG, Gonçalves GK, Corr^ea MS, dos Santos MT. Occlusal characteristics in cerebral palsy patients. J Dent Child (Chic) 2007;74: Rodrigues dos Santos MT, Masiero D, Novo NF, Simionato MR. Oral conditions in children with cerebral palsy. J Dent Child (Chic) 2003;70: Vittek J, Winik S, Winik A, Sioris C, Tarangelo AM, Chou M. Analysis of orthodontic anomalies in mentally retarded developmentally disabled (MRDD) persons. Spec Care Dentist 1994;14: Strodel BJ. The effects of spastic cerebral palsy on occlusion. ASDC J Dent Child 1987;54: Oreland A, Heijbel J, Jagell S. Malocclusions in physically and/or mentally handicapped children. Swed Dent J 1987;11: Jenny J, Cons NC. Establishing malocclusion severity levels on the dental aesthetic index (DAI) scale. Aust Dent J 1996;41: Ballard CF. The effect of lip morphology on the incisors following treatment. Orthod Fr 1969;40: Thomas-Stonell N, Greenberg J. Three treatment approaches and clinical factors in the reduction of drooling. Dysphagia 1988;3: Johnson A. revalence and characteristics of children with cerebral palsy in Europe. Dev Med Child Neuro 2002;44: Bhowate R, Dubey A. Dentofacial changes and oral health status in mentally challenged children. J Indian Soc edod rev Dent 2005;23: Schwartz S, Gisel EG, Clarke D, Haberfellner H. Association of occlusion with eating efficiency in children with cerebral palsy and moderate eating impairment. J Dent Child (Chic) 2003;70: 33-9.